The Cleft Palate Craniofacial Journal

Augmented reality (AR) has recently emerged as a potential alternative to 3D-printed technology in craniomaxillofacial surgery. The purpose of this study was to evaluate the feasibility and accuracy of AR craniotomy guides for fronto-orbital advancement (FOA) compared to conventional 3D-printed guides.

Retrospective comparative study.

Cleft and craniofacial center at a tertiary children's hospital.

3D-printed skull models from 9 patients with metopic or coronal craniosynostosis who underwent FOA between January 2022 and November 2023.

A novel application was developed to project AR craniotomy guides onto 3D-printed skull models. AR guides were compared to conventional 3D-printed guides by utilizing both guidance modalities. The discrepancy at 8 pre-determined reference points was measured, including bilateral nasofrontal (NF), zygomaticofrontal (ZF), barrel stave (BS), and tenon extension (TE).

Measured discrepancy in millimeters between AR guides and 3D-printed guides at the pre-determined reference points.

The anterior reference points (NF, ZF) had mean discrepancies ranging from 0.31 to 0.61 mm. The posterior points had mean discrepancies ranging from 1.39 to 3.28 mm (BS, TE). There was no statistically significant difference found between the two modalities at any reference point.

AR craniotomy guides had a high level of accuracy, particularly at the anterior reference points. AR guides demonstrated feasibility as an alternative to 3D-printed craniotomy guides, in-vitro. Further research is required to translate this novel application to cadaver models and improve precision at posterior landmarks.

To evaluate how patient-reported outcomes change from pre- to post-operation after common procedures in patients with cleft lip and/or palate (CL/P), as well as compared to patients who did not undergo surgery.

Retrospective chart review.

Tertiary care hospital in the United States.

All patients with CL/P who had multiple CLEFT-Q completions from 2021 to 2025.

CLEFT-Q scores.

Totally, 256 patients with 594 CLEFT-Q responses were included. Sixty (23.4%) patients received 66 craniofacial operations between CLEFT-Q completions. Of these operations, 16 (24.2%) were dental extractions/exposures, 15 (22.7%) rhinoplasties, 10 (15.2%) velopharyngeal insufficiency repairs, 6 (9.1%) alveolar bone grafts, 6 (9.1%) orthognathic surgeries, and 4 (6.1%) oronasal fistula repairs. Surgical patients had lower first CLEFT-Q scores across nearly all domains compared to those in the non-surgical cohort (P < .05). Patients who underwent rhinoplasty (n = 15) demonstrated increased face (64.1 vs 49.6, P = .006), nose (64.8 vs 43.3, P = .003), nostrils (62.0 vs 34.2, P = .001), teeth (59.5 vs 41.3, P = .008), and lips (63.5 vs 48.4, P = .007) scores post-operatively. Jaw scores increased following orthognathic surgery, though this did not reach significance (71.2 vs 55.8, P = .100). No other operation had a statistically significant impact on CLEFT-Q scores (P > .05).

Patients who underwent surgery demonstrated improvements in post-operative CLEFT-Q scores compared to patients who did not undergo surgery during this surveyed period. Of those who underwent surgery, rhinoplasty was associated with the most significant improvements in patient facial perception post-operatively.

Maxillary hypoplasia is a common sequela of cleft lip and palate (CLP), often requiring surgical correction through maxillary advancement osteotomy. However, the resulting aesthetic outcome can be unpredictable. Frequently, these cases involve maxillary lateral incisor agenesis, managed either by preserving the space for dental rehabilitation or through orthodontic space closure.

To investigate the correlation between aesthetic and occlusal outcomes and the management of the missing maxillary lateral incisor space. Secondary objectives included assessing the relationship between aesthetic and occlusal scores and space management, nasolabial angle, the extent of maxillary advancement, and the maxillary incisor axis.

Retrospective cohort study.

This retrospective study analyzed 47 patients with CLP sequelae who underwent maxillary advancement surgery between 2008 and 2022 at our center.

Aesthetic evaluation and occlusal evaluation.

Aesthetic evaluation and occlusal evaluation were independently conducted by 5 examiners. Aesthetic and occlusal scores were deemed satisfactory if they exceeded ≥30 of 40 and 15 of 20, respectively. These outcomes were correlated with the preservation of the maxillary lateral incisor space, extent of maxillary advancement, nasolabial angle, and maxillary incisor axis.

No significant improvement in aesthetic or occlusal outcomes was observed, regardless of whether the maxillary lateral incisor space was preserved. However, space closure was associated with greater maxillary advancement (P = .025).

Orthodontic space closure may lead to better outcomes. Further research involving larger sample sizes and consideration of additional factors is needed to determine the optimal approach for each patient.

To describe the perspectives of participants and caregivers of pediatric participants on data sharing in clinical craniofacial research.

Qualitative interview study of research participants’ attitudes about data sharing.

One-to-one interviews via web conferencing.

Adults (n = 7) and adolescents (n = 8) with craniofacial microsomia and caregivers (n = 20) of children with craniofacial microsomia who previously participated in clinical craniofacial research.

Semistructured interview guide addressing 4 main topics: (1) expectations about data collection and sharing; (2) preferences and limitations for data sharing; (3) consent/assent language; and (4) gaps and future needs.

We iteratively developed a qualitative codebook based on inductive interview transcript review and conducted a thematic analysis of coded data.

We identified 5 themes within participants’ descriptions of their attitudes about data sharing: (1) participants hope that research participation and data sharing will advance science for the benefit of the craniofacial community; (2) sharing images is broadly recognized as important for craniofacial research but raises discomfort for some; (3) participants generally view broad data sharing and use positively but raise concerns focused on harm to the craniofacial community; (4) trustworthy researchers and data protections provide reassurance for sharing data; and (5) decisions about pediatric data sharing are complex in the context of developing autonomy.

These findings illustrate ethical complexities for data sharing in clinical craniofacial research related to balancing community-oriented benefits and risks, providing control over sharing images, ensuring researcher trustworthiness, and respecting a child's future autonomy interests.

To describe a pharyngeal bulb reduction program (PBRP) aimed at decreasing the size of the velopharyngeal gap in individuals with repaired cleft palate presenting hypodynamic velopharynx, and to evaluate the effect of the PBRP on pharyngeal bulb dimensions.

Thirteen patients (6 females and 7 males; mean age = 28 years) with repaired cleft palate and hypodynamic velopharynx, who demonstrated normal speech using a pharyngeal bulb, participated in the study. The PBRP lasted two weeks and consisted of sequential pharyngeal bulb reductions during nasoendoscopy combined with intensive speech therapy. After each bulb reduction, measurements of bulb length, width, area, volume, and the prosthesis weight (pre-/post-PBRP) were obtained.

At the end of the PBRP, reductions in bulb dimensions were observed (length: −32.13%, width: −47.26%, area: −61.41%, and volume: −52.80%), as well as a decrease in prosthesis weight (−21.12%).

“In PBRB, the reduction in the dimensions of the pharyngeal bulb, combined with intensive speech therapy, proved effective in stimulating velopharyngeal mechanism movements during speech, leading to a consequent reduction in the size of the velopharyngeal gap. Following the PBRB procedure, reductions in the width and length of the bulb were observed, creating more favorable conditions for performing secondary surgery aimed at correcting velopharyngeal insufficiency.”

To investigate the effects of zinc concentration on palatal development in fetal mice and its association with the aryl hydrocarbon receptor (AhR) signaling pathway.

Pregnant C57BL/6J mice were fed diets with varying zinc concentrations and randomly divided into a zinc-rich (ZR) group, a normal-zinc (NZ) group, and a zinc-deficient (ZD) group. Embryonic development was observed, and the expression levels of AhR signaling pathway-related factors were examined.

No cleft palate was observed in the ZR group or NZ group, whereas the zinc-deficient group exhibited a cleft palate incidence of 27.45%. Hematoxylin and Eosin (HE) staining results revealed failed palatal shelf contact and fusion in the ZD group, whereas complete fusion occurred in the ZR group and a normal medial edge epithelial formed in the NZ group. Results from Immunohistochemistry (IHC), qRT-PCR, and Western blot analyses collectively demonstrated that, compared to the NZ group, the ZR group exhibited significant upregulation (P < .05) in both mRNA and protein expression levels of AhR, HSP90, CYP1A1, and SP1, whereas the ZD group displayed significant downregulation (P < .05).

Zinc concentration is intimately correlated with fetal mouse palatal development, where zinc deficiency may contribute to cleft palate formation through suppression of AhR signaling pathway, whereas zinc-rich conditions facilitate activation of this pathway. This study reveals the regulatory role of zinc-AhR signaling axis in palatal development through mouse models, offering novel theoretical insights into zinc deficiency-induced cleft palate pathogenesis and establishing a foundational framework for preventive interventions.

To verify the relationship between nasal function, nasal patency, respiratory mode, and type and lung function through lung capacity in patients with cleft lip and palate (CLP).

Quantitative and cross-sectional clinical research.

Tertiary-level craniofacial hospital.

Thirty patients between 18 and 30 years old with repaired CLP and presence of dentofacial deformity were evaluated.

The respiratory mode and type were determined through perceptive assessment and obtaining respiratory symptoms, through a self-reported questionnaire. Spirometry and rhinomanometry were applied.

Forced vital capacity and peak forced expiratory flow.

It was identified that the respiratory mode directly influences peak expiratory flow (PEF) values, as well as the presence of respiratory disease. Forced vital capacity (FVC) showed a statistically significant relationship (p = .036), decreasing its values with the presence of self-reported nasal obstruction. By rhinomanometry, an average of 0.537 (±140 cm²) was obtained for nasal cross-sectional area values, with only 7.69% of cases with values below 0.400 cm², suggesting nasal obstruction.

There is a relationship between nasal and pulmonary function in patients with CLP, with regard to the respiratory mode, presence of respiratory disease and self-reported nasal obstruction, influencing lung capacity values such as FVC and PEF.

This study aimed to assess the impact of two-stage palatoplasty incorporating a vomer flap on facial growth of individuals with bilateral cleft lip and palate (BCLP).

Hospital for Rehabilitation of Craniofacial Anomalies, University of São Paulo, Bauru, Brazil.

A cross-sectional study involving patients who underwent labial adhesion and anterior palatoplasty with a vomer flap at 3 and 5 months, followed by posterior palatoplasty at 12 months. Individuals without prior orthodontic treatment or secondary alveolar bone grafting were included. Cephalometric radiographs were obtained during mixed or early permanent dentition. A control group of untreated class I individuals without clefts was evaluated.

The sample comprised 49 individuals with BCLP (17 females, 32 males; mean age 8.91 years) and 44 control subjects (17 females, 27 males; mean age 8.91 years).

Cephalometric analysis was conducted using Dolphin Imaging software (version 11.95). Intergroup comparisons were performed using independent t-tests and Mann-Whitney U tests (P < .05).

No significant difference between groups was found in maxillary sagittal position. However, BCLP group showed a significantly smaller SNB angle (mean difference −2.1°, P < .003) and a more convex skeletal profile compared to the noncleft group. Additionally, BCLP group exhibited severe lingual inclination of the maxillary incisors, (MD = –31.6°, P < .001) and a high prevalence of anterior crossbite.

At the time of observation, the maxillary sagittal position appeared comparable between groups. However, mandibular retrusion contributed to a more convex profile. The severe retroinclination of maxillary incisors determined a negative overjet. Longitudinal follow up are needed to confirm long-term maxillofacial growth outcomes.

To assess the prevalence of obstructive sleep apnea (OSA) in a cohort of children with craniofacial syndromes (CFS) within a single institution, and to describe their polysomnographic (PSG) characteristics and the overall management of OSA in this cohort.

A single-center retrospective study. Electronic healthcare records were used to access information regarding medical history, surgical history, and PSG details of patients with CFS.

This study took place at a tertiary care center affiliated with a school of medicine.

Patients with CFS under the age of 18 years with PSG data were assessed.

No intervention was performed as it was retrospective study.

Obstructive sleep apnea prevalence among children with CFS.

A total of 46 patients were included in this study. In our study, 69.6% of the children with CFS had OSA, with 39.1% of them classified as severe. We performed mandibular distraction osteogenesis (MDO) to treat 26.5% of patients with OSA. The prevalence of OSA was 93.8% among a total of 16 Pierre Robin Sequence (PRS) patients. MDO substantially decreased apnea-hypopnea index (AHI), rapid eye movement AHI, and EtCO2 in PRS patients while simultaneously increasing SpO2 and total sleep time.

Obstructive sleep apnea is more prevalent in children with CFS than was previously recognized, and MDO is the most frequently employed treatment. Pierre Robin Sequence is the most prevalent pediatric CFS associated with OSA. Mandibular distraction osteogenesis significantly improves OSA and sleep quality in PRS patients.

To clarify how the preoperatively determined position of the Cupid's bow peak on the lateral lip element affects postoperative nasolabial formation in patients with unilateral cleft lip. We hypothesize that setting the proposed peak of the Cupid's bow close to the nostril floor causes the cleft-side nasal alar base to droop while positioning it near the oral commissure shortens the red lip, causing an elevated cleft-side oral commissure.

Retrospective study.

Single institution.

Twenty-seven patients with non-syndromic complete unilateral cleft lip and palate (UCLP) who received primary lip plasty at our department.

Primary lip repair for patients with UCLP.

We tested our hypothesis using three-dimensional facial images taken before and 1 year after primary lip repair. Bilateral measurements included lip height (distance between the nasal alar base and the Cupid's bow peak), red lip length, and the vertical height of the nasal alar base and oral commissure.

Correlation analysis showed a significant relationship between preoperative cleft-side lip height and the postoperative position of the cleft-side nasal alar base (P = .0024, Pearson's correlation analysis). Preoperative short red lip remained short afterwards (P = .000036, Pearson's correlation analysis), causing a lip cant with an elevated cleft-side oral commissure (P = .012, Pearson's correlation analysis).

Postoperative nasolabial formation is influenced by the preoperatively proposed position of the Cupid's bow peak. This study provides a comprehensive principle explaining the relationship between the location of the proposed Cupid's bow peak on the lateral lip elements and postoperative nasolabial formation in primary lip repair in patients with unilateral cleft lip.

Enhanced Recovery After Surgery (ERAS) protocols are evidence-based perioperative management pathways designed to optimize surgical outcomes. The American Society of Craniofacial Surgeons (ASCFS) Presidential Task Force has developed a series of ERAS protocols for patients with cleft and craniofacial anomalies. We outline an ERAS protocol for secondary alveolar bone grafting using cancellous bone graft from the iliac crest for patients with cleft lip and palate.

The authors extracted information from existing peer-reviewed literature and our institutional experience at a large, tertiary pediatric hospital through retrospective chart review to guide surgeons in the pre-hospitalization, preoperative, intraoperative, and postoperative phases of care of alveolar bone grafting.

In the pre-hospitalization phase, our ERAS protocol emphasizes family education and expectation management, as well as minimization of preoperative fasting. In the preoperative phase, oral midazolam is recommended to reduce patient anxiety. In the intraoperative phase, we emphasize multimodal pain control with regional nerve blocks, bupivacaine-soaked absorbable sponge in the iliac crest, and ketorolac to minimize postoperative narcotic use. To prevent postoperative nausea and vomiting, we emphasize the use of an oropharyngeal pack prior to incision and nasogastric tube evacuation of the gastric contents at the completion of surgery, as well as a combination of ondansetron with dexamethasone intraoperatively. In the postoperative phase, we recommend dexmedetomidine, early postoperative oral feeding and hydration, and early ambulation with Physical Therapy consultation.

The present study sought to outline an ERAS protocol for secondary alveolar bone grafting in pediatric patients with cleft lip and/or palate to optimize surgical outcomes.

To assess the demographic patterns, surgical volume, and COVID-19 effect on cleft lip and/or palate (CL/P) patients at a mission-based hospital, providing insight into the burden of cleft care and the role of global outreach in resource-limited settings.

Retrospective cohort study.

Social aid hospital in Guatemala.

Patients undergoing CL/P surgery between July 2014 and December 2024.

Surgical missions for CL/P.

Primary outcomes included surgical volume, geographic distribution of patients, and age at primary surgery.

Over 10 years, 2010 CL/P patients were treated by 98 surgeons. Patients ranged from 11 months to 73 years (mean age 7.4 years); 64% were male. The most common conditions were unilateral cleft lip (38.4%), cleft palate (28.2%), and cleft palate with unilateral cleft lip (19.2%). Patients came from 18 of Guatemala's 22 departments. Surgical volume declined during the pandemic but rebounded in 2021, reaching the highest volume of patients treated. The average age at primary surgery decreased significantly over time, from 9.9 years pre-COVID to 4.9 years during the pandemic and 3.8 years post-COVID (P < .001).

Our institution provides essential cleft care to underserved populations across Guatemala. Even with disruptions caused by the pandemic, surgical delivery continued to improve. These findings reflect the hospital's ability to adapt and illustrate how mission-based models can continue improving timely surgical access over time. Efforts to further reduce treatment delays and expand follow-up care remain essential to optimizing cleft outcomes.

Intraoperative transfusion during corrective surgery for craniosynostosis is common. We sought to evaluate determinants of intraoperative transfusion for patients with nonsyndromic, single-suture craniosynostosis undergoing primary repair.

We queried the multicenter Pediatric Craniofacial Surgery Perioperative Registry for patients undergoing correction of craniosynostosis between June 2012 and January 2023. Nonsyndromic patients who underwent primary repair of single suture craniosynostosis at <12 months of age were included. Univariate and multivariate logistic regressions were performed.

Intraoperative transfusion requirement.

A total of 3994 patients met inclusion criteria, and 2261 (56.6%) received an intraoperative blood transfusion. Those who received an intraoperative transfusion had longer surgery duration (93.8 ± 1.5 vs 180.1 ± 1.9 min, P < .001), increased weight (6.8 ± 0.04 vs 7.8 ± 0.03 kg, P < .001), and were older (4.1 ± 0.1 vs 6.6 ± 0.1 months, P < .001) and more likely female (odds ratio [OR] 1.18, 95% confidence interval [CI]:1.03-1.35, P = .018). For 144 (3.6%) patients who received preoperative iron supplementation, the incidence of intraoperative transfusion was decreased (OR 0.39; 95%CI: 0.27-0.57, P < .001). In multivariate analysis, factors independently associated with higher odds of intraoperative transfusion included longer surgery duration (P < .001), increasing age (P = .017), lower weight (P = .011), higher American Society of Anesthesiologists class (P < .001), procedure (P < .001) (especially nonendoscopic), lack of preoperative iron supplementation (P < .001), and lower preoperative hemoglobin (P = .038).

Various factors influence the incidence of transfusion in patients undergoing primary surgical correction of single suture craniosynostosis. Timing of surgery and optimization of preoperative hemoglobin with iron supplementation represent possible modifiable risk factors that warrant additional, prospective study.

This study aimed to evaluate the characteristics and risk factors associated with microtia at Airlangga University Hospital.

A retrospective and unmatched case–control study involving 354 microtia patients and 354 controls, using convenience sampling from regions across western to eastern Indonesia.

Airlangga University Hospital, a national referral center for microtia in Indonesia.

Patients and controls were recruited through online questionnaires.

None; data were collected using Google Forms.

Prevalence, laterality, severity (Hunter's Classification), and potential risk factors, including parental health, smoking exposure, folic acid intake, TORCH vaccination, and family history.

Microtia was more prevalent in males (78%) with a male-to-female ratio of 3.53:1 (P < .001). Unilateral cases dominated (80.2%), mainly affecting the right ear (61.3%). Grade 3 microtia was most common (59%). After performing multiple unconditional logistic regression analyses, significant risk factors associated with the microtia group included maternal history of miscarriage/stillbirth (adjusted odds ratio [AOR] = 4.74-4.96), diabetes (AOR = 6.46), hypertension (AOR = 4.18), maternal smoking (AOR = 2.06), paternal smoking (AOR = 2.42-2.83), lack of TORCH vaccination (AOR = 1.59-2.02), and family history (AOR = 5.36), all with P value < .05.

Microtia in Indonesia reflects global patterns, with male predominance and right-sided unilateral cases. Associated risk factors include parental smoking, maternal comorbidities, lack of TORCH vaccination, and genetic predisposition. Public health strategies should promote smoking cessation, prenatal care, and vaccination. Further studies should ensure matching between the control and microtia groups, minimize recall bias, and explore genetic links.

To map and synthesize evidence on the relationship between socioeconomic status (SES) and orofacial clefts (OFC), identifying key SES indicators, study designs, and evidence gaps to inform future research and policy.

Scoping review following Arksey and O’Malley's framework and reported per PRISMA-ScR guidelines.

Systematic searches of PubMed, Scopus, and Web of Science with no date or language restrictions.

Peer-reviewed studies of individuals with non-syndromic OFC that explicitly measured at least one SES indicator (income, education, occupation, insurance status, area-level deprivation index, or composite SES index).

Not applicable.

Identification and charting of SES measures, study characteristics (design, setting, sample size), and domains of SES–OFC associations (eg, access to care, OFC risk).

Of 259 retrieved records, 42 studies met inclusion criteria. Publications increased over time, with 78.6% conducted in high-income countries (50.0% in North America). Retrospective designs predominated (59.5%). The most common SES indicators were demographic factors (61.9% of studies; race/ethnicity in 52.4%) and household income (42.9%). Two principal domains emerged: SES impact on access to multidisciplinary OFC care (45.2% of studies; all reporting greater barriers for low-SES groups) and SES as a risk factor for OFC (33.3%; most finding increased risk with lower SES).

SES research in OFC is expanding but remains concentrated in high-income settings with diverse measures. Harmonized core indicators—LMICs included—and longitudinal, registry-based studies are needed. Findings support targeted policy interventions (eg, subsidies, resource allocation) and routine SES screening in OFC care pathways to promote equitable access.

To assess the relationship between neighborhood-level social disadvantage, as measured by Area Deprivation Index (ADI) and Childhood Opportunity Index (COI), and postoperative outcomes and follow-up care after primary or secondary cleft palate repair.

Retrospective cohort study.

Academic tertiary care center.

Pediatric patients with cleft palate with or without cleft lip (CP ± L).

Primary palatoplasty, revision palatoplasty, or closure of oronasal fistula.

Postoperative complications and clinic follow-up adherence.

A total of 244 patients were included in the study. Among all patients, there was only a moderate correlation between patients’ ADI and COI quintiles (R² = 0.465, P < .001). In patients undergoing primary palatoplasty, greater ADI neighborhood deprivation was significantly associated with a greater number of no-show (P = .011) and cancelled (P = .025) clinic appointments. In patients undergoing revision palatoplasty, greater ADI neighborhood deprivation was significantly associated with higher rates of postoperative complications (P = .016) and a greater number of no-show clinic appointments (P = .036). Greater COI disadvantage was associated with significantly higher rates of hospital readmission (P = .049) and fewer cancelled clinic appointments (P = .049) in revision palatoplasties. Neighborhood disadvantage was not predictive of any oronasal fistula repair postoperative outcomes, follow-up adherence, or patient characteristics at surgery.

Neighborhood-level social disadvantage indices are predictive of suboptimal surgical outcomes and follow-up adherence in patients undergoing cleft palate surgery and could be used to identify patients who may benefit from additional clinic outreach and support.

There is no universal protocol for extubation following mandibular distraction osteogenesis (MDO) surgery in infants with Robin sequence (RS). The aims of this study were to identify the frequency and contributing factors for reintubation after MDO, to help determine the optimal setting for planned extubation.

This is a retrospective observational study of patients with RS managed with MDO during their first year of life from 2013 to 2021.

The primary outcome variable was the need for reintubation <24 h after extubation. A secondary outcome was the frequency of oxygen saturation <95% after extubation.

Fifty-two subjects were included. Of these, 31 (59.6%) were male, 43 (82.7%) had a cleft palate, 19 (36.5%) were syndromic, and 24 (46.2%) had ≥1 comorbidity. Extubation was at 3.6 ± 2.4 days after surgery and 1 patient (1.9%) required reintubation. Forty-one (78.8%) had ≥1 transient oxygen desaturation managed with supplemental oxygen (n = 25, 61%) or continuous positive airway pressure (n = 16, 39%). Increased risk for postextubation respiratory events was associated with Stickler syndrome (P = .01), musculoskeletal, neurologic, or endocrine comorbidities (P < .001), low birthweight (P = .044), and high preoperative obstructive Apnea-Hypopnea Index (P = .037).

Reintubation was rare and minor postextubation oxygen desaturations, while common, were readily treated utilizing standard intensive care unit protocols. We conclude that most infants with RS can be safely extubated in their care unit following MDO. This minimizes medical resources, decreases hospital charges, and simplifies patient management compared to returning to the operating room for extubation.

This study aims to analyze the correlation between orofacial cleft (OFC) birth prevalence and residential altitude from a global perspective, providing more evidence for the development of early screening and prevention policies for OFC, particularly in high-altitude regions.

The birth prevalence of OFC, summary exposure value of associated risk factors, and Socio-demographic Index data were extracted from the Global Burden of Disease Study 2021. Geographic altitude (GA) and global altitude-population data were extracted from published data. A novel Population Altitude Index (PAI) was developed to describe the altitude at which a country's population is primarily concentrated. Spearman correlation coefficient (R_s), multinominal logistic regression, partial correlation analysis, Mann-Whitney U test, and paired t test were employed to analyze the relationships between OFC birth prevalence, GA, PAI, and other related factors. Sensitivity analysis was performed using the generalized linear model. The population attributable fraction was used to assess the contribution of high residential altitude to the birth prevalence of OFC.

Mann-Whitney U test and paired t test indicated a statistical difference between GA and PAI (P < .05). Orofacial cleft birth prevalence showed a moderate positive correlation with PAI (R_s = 0.31, P < .05), stronger than with GA (R_s = 0.21, P < .05). Partial correlation analysis, multinominal logistic regression, and generalized linear model confirmed PAI's independent association with OFC birth prevalence after adjusting for other risk factors. A PAI threshold of 375 m yielded the largest intergroup difference (Cohen d = −0.65). The estimated population attributable fraction for OFC birth prevalence in regions with a PAI >375 m was 9.9% (lower: 4.7%, upper: 14.1%).

Residential altitude, particularly when assessed by PAI, is positively associated with national and regional level OFC birth prevalence. The PAI may serve as a valuable index for future altitude-related epidemiological studies.

The German system of care for patients with cleft lip and/or palate (CL/P) is fragmented. An argument against centralization is that the higher burden of travel for patients in remote and rural areas would reduce appointment adherence and subsequent care outcomes. Our objective was to test the association of travel burden and adherence and outline potential benefits of centralized cleft care.

Follow-up appointments June 2005 to August 2020 were retrospectively analyzed for patients with CL/P in a large German tertiary care center. Distance to care center and travel time by car/public transportation were calculated using Google Maps. Demographic characteristics included population density, degree of urbanization, and average disposable income per capita of the municipality of origin. Multiple regression models including flexible spatial function learning assessed associations with follow-up appointment adherence.

Patients (n = 1140) had 9447 scheduled appointments with a 48.2% attendance rate. Distance to the clinic (M = 85.19 ± 75.12 km, range 0.45–536), travel time (M = 71.18 ± 48.5 min, range 2–330), population density (725.32 ± 807.82 inhabitants/km², range 14–4861), and lower average income (20,993.38 ± 1220.72 Euro, range 14,521–32,348.83) were not associated with appointment attendance. Patients from more rural areas were slightly more likely to attend check-ups (p = 0.027, 95% CI [0.016, 0.212]).

The burden of travel to the clinic was not associated with attending follow-up appointments for patients with CL/P. These findings challenge a common assumption about attendance and warrant further multi-center studies to inform policy decisions for restructuring cleft care in Germany.

This study compared anxiety between Chinese caregivers of children with non-syndromic cleft lip (CL) or cleft palate (CP) at the time of their first surgery and control group as well as identifying factors associated with caregiver anxiety.

Retrospective, observational cohort study.

Department of Cleft Lip and Palate in West China Hospital of Stomatology, Sichuan University.

Caregivers of children under 3 years old.

The 7-item Generalized Anxiety Disorder Scale (GAD-7).

Caregivers were mostly married mothers aged 19-35 years in the control (n = 131), CL (n = 132), or CP (n = 147) groups. Mean GAD-7 scores were 4.90 ± 4.53, 3.74 ± 3.95, and 4.82 ± 4.64 for the control, CL, and CP groups, respectively. The only differences were lower anxiety for the CL group compared to controls (P = .028) and to the CP group (P = .038). Most caregivers reported no anxiety (control 50.38%, CL 65.15%, CP 53.74%) or mild anxiety (control 39.69%, CL 27.27%, CP 31.29%). More CL caregivers exhibited no anxiety compared to controls (65.15% vs. 50.38%), while more CP caregivers exhibited moderate anxiety compared to controls (12.24% vs. 3.82%). Higher GAD-7 scores were associated with having fewer children for CL caregivers, and with younger age and lower education for CP caregivers.

At the time of first surgery, most Chinese caregivers of children with CL/CP had no to low anxiety. Additional psychosocial support may benefit the 8% to 15% of caregivers of children with a cleft who had moderate or severe anxiety.

In low resource settings, there are several barriers to achieving a multidisciplinary approach covering all aspects of cleft including psychosocial services. The study aim was to gain insight into current psychosocial practices in these settings.

Cross-sectional study.

Comprehensive Cleft Care Workshop in October 2024.

Workshop Attendees.

A 20-item survey including quantitative and qualitative questions about psychosocial practices in participants’ workplaces was distributed.

The main outcomes were to assess cleft psychosocial practices, and the challenges faced when integrating psychosocial care in low resource settings.

Seventy-six respondents were working in low-resource settings in Africa or Asia, with 67.2% working in plastic or oral/maxillofacial surgery department and 68.4% working in an academic hospital. Almost 90% of respondents work in a multidisciplinary team. Of those who responded that psychosocial care was available in their setting (n = 44), 47.7% reported that either psychologists or psychiatrists were responsible for providing this care. Twenty-five (56.8%) of these respondents reported that although psychosocial support is available, this service is not provided by a cleft specialist. Most respondents reported a lack of resources/tools (68.4%) as a challenge to referring patients to psychosocial care and mentioned a need for: (1) more cleft-specialized psychosocial personnel; (2) dedicated financial budgets; (3) increased awareness; and (4) protocols to integrate multidisciplinary discussions.

Availability, accessibility and knowledge of the importance and recommended delivery of psychosocial services in cleft remains limited in low resource settings. Further research into psychosocial needs and ways of improving delivery is required.

Prior work has identified numerous barriers to standardized outcome measurement in cleft care. The objective of this study was to evaluate a multifaceted implementation strategy to collect standardized outcomes for children with cleft lip and/or palate (CL/P).

A one-armed, single-site pilot study of a multifaceted implementation strategy.

A metropolitan children's hospital.

Five-year-old children with CL/P.

A multifaceted implementation strategy for standardized outcome measurement.

The primary outcome measure was penetration of the intervention, defined as the proportion of children for whom the standardized measures of aesthetic, dental, and speech outcomes were collected. Penetration pre-implementation and 12 months post-implementation were compared. The secondary outcome was acceptability of standardized outcome measurement among providers and staff, evaluated with the Acceptability of Intervention Measure.

The implementation strategy resulted in high penetration of standardized outcome measurement: measurement of aesthetic outcomes increased from 7% to 82% (p < .0001); measurement of dental outcomes increased from 0% to 56% (p < .0001); measurement of speech outcomes increased from 29% to 77% (p = .0001). Acceptability of standardized outcome measurement was high, with 93% of cleft team providers and staff (n = 14/15) reporting that they somewhat or completely agreed that standardized outcome measurement met their approval, was appealing, was welcomed, and was liked. Penetration continued to improve after the implementation period, with high rates of outcome measure collection sustained for four years following implementation.

Deployment of a multifaceted implementation strategy led to substantial increases in the collection of aesthetic, dental, and speech outcomes among children with CL/P.

To evaluate morphologic outcomes following mature cleft rhinoplasty using exclusive septum cartilage grafting.

Retrospective, observational study.

Single institution, 8-year retrospective review.

A total of 31 facially mature patients with non-syndromic unilateral cleft lip were included in this study.

Patients underwent mature cleft rhinoplasty using septum cartilage as the exclusive graft source.

Preoperative and postoperative photographs (at least 1 year after surgery) were analyzed using 8 distinct anthropometric parameters to assess for statistical significance in the changes to nasal morphology. Paired-samples t-tests were performed, and statistical significance was defined as P < .05.

The mean changes following mature cleft rhinoplasty were: alar cant 1.96 to 0.95 (P < .001), alar height angle 1.90 to 1.21 (P = .006), nasal tip deviation 2.90 to 1.99 (P < .001), vertical alar height ratio 0.95 to 0.95 (P = .4), nasofacial angle 28.52 to 31.48 (P = .003), nasolabial angle 83.15 to 87.96 (P = .01), columellar angle 9.09 to 3.30 (P < .0001), and nostril height ratio 0.80 to 0.88 (P < .001).

The nasal septum can be considered as a reliable initial choice for cartilage grafts during mature cleft rhinoplasty.

To determine whether human milk feeding in children with cleft palate (CP ± L) reduces tympanostomy tubes and improves hearing, speech, and language outcomes.

Retrospective cohort study.

Tertiary children's hospital at a Cleft Craniofacial Specialty Clinic.

319 children with CP ± L who were born between April 2005 and April 2015 were included.

Impacts on otological, hearing, and speech/language outcomes were analyzed in children fed human milk for any duration, ≥3 months, and ≥6 months.

Tympanostomy tube placement history, hearing status before and after tube placement, and speech/language development at 2 and 5 years of age.

Children with CP ± L fed human milk for ≥3 months (143/319, 44.8%) had decreased odds of abnormal hearing tests (odds ratio [OR]: 0.174, 95% confidence interval [CI]: 0.0266-0.845) or tympanograms (OR: 0.059, 95% CI: 0.008-0.936) before tube placement and decreased odds of bilateral effusion at tube placement (OR: 0.236, 95% CI: 0.089-0.628), even when accounting for Robin sequence and insurance type. There was no significant association between human milk feeding and hearing after tube placement. However, language delays (OR: 0.279, 95% CI: 0.084-0.930) and speech sound production disorder (OR: 0.488, 95% CI: 0.246-0.967) at 5 years of age were less common in those who received human milk of any duration.

Feeding human milk to children with CP ± L was associated with improved otological, hearing, speech, and language outcomes. Despite the known challenges of feeding infants with CP ± L, this study demonstrates the added benefits of human milk in this population.

Achieving facial symmetry in patients with Tessier no. 7 cleft undergoing oral commissure reconstruction remains a challenge, with variable aesthetic and functional outcomes. Due to the rarity of this condition, data on long-term treatment effectiveness is limited. This study evaluates postoperative facial symmetry using facial anthropometric analysis in patients who underwent commissuroplasty over a 19 year period.

Retrospective cohort study.

Single-institution craniofacial center.

Patients who underwent commissuroplasty for Tessier no. 7 cleft between 2005 and 2024.

Myomucosal advancement flap commissuroplasty technique.

Facial symmetry was assessed using 5 2-dimensional measurements: stomion-to-chelion (st_ch), chelion-to-ala (ch_al), chelion-to-exocanthion (ch_ex), chelion-to-pogonion (ch_pg), and commissure angle (co_ang). A symmetry ratio of 1.0 indicated ideal symmetry. Major revisions included secondary surgery for asymmetry or scar revision.

Thirty-two patients were included; 24 had unilateral (76% right-sided) and 8 had bilateral clefts. Median age at surgery was 8.1 months; 48% were female. Additional Tessier clefts were present in 25% of patients, and 25% had syndromic diagnoses. Major revisions were required in 28.6% of cases. Symmetry significantly improved in 4 of 5 facial regions, though commissure angle showed no significant change. Symmetry ratios remained stable over a median follow-up of 3.3 years.

Commissuroplasty improves facial symmetry across most parameters in patients with Tessier no. 7 cleft, with durable long-term outcomes. Limited improvement in commissure angulation highlights the need for continued refinement of surgical techniques.

To determine the effect of early hard palate closure by means of a Vomer flap (VF) on the nutritional status of children with cleft lip and palate.

Multicenter retrospective cohort study.

Three referral centers for oral clefts.

Nonsyndromic children with UCLP or BCLP.

Interventions: Early hard palate closure with a VF at cheiloplasty around 3 months versus isolated cheiloplasty, both followed by palatoplasty around 9 to 10 months.

Anthropometric measures (weight and height) and related weight-for-age and height-for-age Z-scores at the time of cheiloplasty and palatoplasty.

Between 2012 and 2024, 122 children with UCLP or BCLP underwent cleft repair surgery. Median age at cheiloplasty was 111 (99-126) days and 304 (284-342) at palatoplasty. In 60% of them (n = 73), a VF was performed simultaneously with cheiloplasty. Weight increase between cheiloplasty and palatoplasty was slightly more for the VF group compared to the control group by 0.24 kg (SE = 0.15, P = .13). No height difference was measured between the groups (P = .80). Z-score variation between cheiloplasty and palatoplasty was significantly different for all study groups, indicating a strong normalization of the growth curve after cheiloplasty (P < .001).

Early hard palate closure using a VF may favor growth in children with cleft lip and palate. The deviation from the normative growth curve of infants decreases significantly between cheiloplasty and palatoplasty, regardless of whether a VF was performed.

To compare pharyngeal airway changes after anterior maxillary distraction and conventional maxillary distraction in patients with unilateral cleft lip and palate (UCLP).

Retrospective study.

Tertiary care medical center for cleft care.

A total of 20 patients with UCLP and hypoplastic maxilla were selected.

Group 1 (n = 10): Anterior Maxillary Distraction, Group 2 (n = 10): Conventional maxillary distraction.

The primary outcomes assessed on pre- and postsurgical cone beam computed tomography were changes in the nasopharyngeal, oropharyngeal, and total pharyngeal airway area and volume, as well as the minimal axial area. These 2 distraction techniques were compared for their impact on upper airway dimensions.

In Group 1, the nasopharyngeal area increased significantly by 43.60 mm², and the oropharyngeal area decreased by 40.80 mm². There were no significant changes in the volume and area of all the other pharyngeal parameters. In Group 2, the nasopharyngeal area and volume increased by 202.70 mm² and 5.0 cm³, respectively, the oropharyngeal area and volume increased by 176.20 mm² and 7.9 cm³, respectively, the total pharyngeal area and volume increased by 380.50 mm² and 14.0 cm³, respectively, and the minimal axial area increased by 133.50 mm².

Anterior maxillary distraction had a negligible impact on the pharyngeal airway compared to conventional distraction. The choice between these 2 interventions should also take into consideration associated comorbidities related to pharyngeal airway and speech.

The objective of this study was to systematically analyze the cleft lip and/or palate (CL/P) literature published between 2000 and 2025 to identify dominant research themes, temporal trends, and potential gaps by applying a natural language processing-based topic modeling approach.

We analyzed articles retrieved from the Scopus database and identified thematic clusters using the BERTopic algorithm, which integrates BERT embeddings, Uniform Manifold Approximation and Projection dimensionality reduction, and Hierarchical Density-Based Spatial Clustering of Applications with Noise clustering.

This study exclusively examined articles related to CL/P.

This study involved no patients or participants.

Not applicable as this is a BERTopic-based analysis.

Outcomes included identifying dominant research topics, trend analysis, distribution across countries and journals, and classifying topics as “hot” or “cold” based on temporal trends.

We identified 29 thematic clusters. The most common topics included “Velopharyngeal Dysfunction and Fistula Repair,” “Nonsyndromic Cleft Lip and Palate and Genetic Associations,” and “Unilateral Cleft Lip and Palate and Maxillofacial Asymmetry.” “Quality of Life and Psychosocial Impact in Children,” “Surgical Repair Approaches,” and “Presurgical Nasoalveolar Molding Techniques” emerged as “hot topics.” The United States led in publication volume, while certain topics dominated in specific countries. The Cleft Palate-Craniofacial Journal published the most articles.

This study provides a comprehensive thematic map of CL/P research, highlighting strengths and underexplored areas. BERTopic offers an efficient, scalable method for analyzing large-scale scientific literature and identifying future research priorities.

This systematic review and meta-analysis aimed to quantify morphological changes in the anterior maxillary arch—specifically anterior cleft width (CW) and anterior curvature angle (ACA)—following primary lip repair in children with unilateral cleft lip and palate (UCLP).

A comprehensive search of PubMed, Google Scholar, DOAJ, and the Cochrane Library was conducted through January 2025. Eligible studies reported quantitative pre- and post-operative data on anterior arch morphology. Pooled effect sizes were calculated using a random-effects model with the Knapp–Hartung adjustment. Meta-regression assessed the influence of baseline cleft width and follow-up duration.

Ten studies comprising 11 patient cohorts and 265 participants met the inclusion criteria. The pooled mean difference in anterior CW) was −6.217 mm (95% confidence interval (CI): −7.230 to −5.204; P < .001), with substantial heterogeneity (I² = 81.8%). Meta-regression identified initial cleft width as a significant predictor (β = −0.842; P = .002), and follow-up duration showed a borderline association (β = −0.387; P = .051). The predictive equation was: CW(change) = 4.465 − 0.842 × initial width − 0.387 × duration. Lip repair also reduced the anterior curvature angle (MD = −14.66°; 95% CI: −19.62 to −9.7), though heterogeneity was very high (I² = 88.1%).

Primary lip repair significantly improves anterior arch dimensions in children with UCLP. Initial cleft width predicts postoperative changes, and the proposed model may support early treatment planning. However, due to substantial heterogeneity across studies, small sample sizes in some included cohorts, and very low certainty of evidence for both outcomes (GRADE approach), the findings should be interpreted with caution and considered exploratory.

Optimal timing for primary cleft lip (CL) repair remains controversial due to a paucity of literature reporting outcomes. This study sought to identify trends in timing of CL repair over a 10-year period.

Cross-sectional analysis of the National Surgical Quality Improvement Program-Pediatrics (NSQIP-P) between 2013 and 2022.

CL patients undergoing primary CL repair were divided into 4 cohorts based on age (months) at repair: 0 to 3, 3 to 6, 6 to 9, and 9 to 12.

Univariable statistics, multivariable regressions, and trend analyses were conducted to identify predictors for operation at latter ages and to evaluate trends over time.

11,585 CL patients were identified with the majority undergoing repair between 3 and 6 months of age (63%). White and Asian patients predominately underwent repair between 0 and 3 months (68% and 5%, respectively), while African American and Hispanic patients underwent repair between 9 and 12 months (12% and 22%, respectively, P < .001). On regression analysis, patients with cardiac risk factors (CRF), asthma, bronchopulmonary dysplasia, developmental delay, CNS abnormalities, and nutritional supplementation (P < .001) were more likely to be repaired at later time intervals. Over the past decade, Hispanic and Asian patients undergoing CL repair increased between 0 and 3 (β = 0.06) and 3 and 6 (β = 0.05) months of age, respectively. Patients with CRF and airway abnormalities also significantly increased between 0 and 3 months.

The present study identified an increase in non-White patients and patients with severe comorbidities undergoing early CL repair over a 10-year period. Healthcare professionals should note these trends when treating their own patient populations.

To establish correlations between skeletal jaw relationship measured on lateral cephalograms and Goslon Yardstick scores for dental arch relationship (DAR) on orthodontic study models for unilateral cleft lip and palate (UCLP).

Retrospective review of consecutive cases.

Multidisciplinary cleft and craniofacial clinics at two tertiary care centers in the Western Cape, South Africa.

Forty-nine consecutive patients with nonsyndromic UCLP before they received orthodontic treatment and secondary alveolar bone graft (SABG).

Fourteen cephalometric angles measured by two observers and Goslon Yardstick scores determined by three observers. Inter- and intraobserver reliability determined using Cohen's Weighted Kappa statistic.

Age, gender, and side of cleft were recorded. Cephalometric measurements and Goslon scores compared with regression analysis to determine correlations between angle ANB (cephalometric angle indicating anteroposterior relationship between the maxilla and mandible) and Goslon scores.

Mean age 10.7 years; 22 males and 27 females. Thirty-four (69.4%) of the clefts were left-sided. Kappa statistics ranged from good to very good for inter- and intraobserver reliability for cephalometric measurements and Goslon scores. No statistically significant differences between genders for cephalometric measurements and Goslon scores (P > .05). Mean ANB = 0.2(2.39) indicates Class III skeletal relationships for these individuals. Mean Goslon score 2.89. There was a moderate negative correlation of r = −0.5691 (P = 0) between ANB and Goslon score.

Moderate negative correlation between ANB and Goslon Yardstick provides evidence that Goslon scores are valid and reliable indicators of skeletal jaw discrepancy for UCLP without the errors encountered using cephalometric radiographs.

The presence of a bony palatal bridge at the time of alveolar cleft grafting has been shown to improve bone graft outcomes. The purpose of this study was to determine if there is an association between palate repair with a vomer flap and formation of a bony palatal bridge in patients with cleft lip and palate.

Single-institution, retrospective cohort study.

Tertiary children's hospital in an urban setting.

Patients with cleft lip and palate who underwent primary palatoplasty between 1996 and 2017 and had cone beam computed tomography (CBCT) prior to alveolar bone grafting.

Primary palatoplasty with or without vomer flap.

The presence of a bony palatal bridge on pre-alveolar bone graft CBCT.

Of 238 patients who underwent primary palatoplasty at a mean age of 10.0 months, 225 (94.5%) had a vomer flap. Bony palatal bridge formation was observed in 128 patients (53.8%) on pre-graft CBCT scans. Among patients with a bony palatal bridge, 121 (94.5%) had a vomer flap at primary palatoplasty. No significant association was found between use of a vomer flap and bony palatal bridge formation (p = 0.996). Secondary analyses demonstrated no significant associations with age at primary palatoplasty (p = 0.157), history of pre-surgical orthopedics (p = 0.777), syndromic status (p = 0.101), or cleft type (p = 0.408).

This study demonstrated that the use of a vomer flap during primary cleft palate repair was not associated with the formation of a bony palatal bridge. Further studies will focus on other factors that may contribute to the etiology of bony palatal bridge formation.

To describe a rare complication of supernumerary tooth displacement into the nasal cavity and subsequent migration to the gastrointestinal tract in a patient with cleft lip and palate.

Case report and literature review.

Hospital-based care at the Center for Comprehensive Care of Cleft Lip and Palate patients.

A 10-year-old male with a unilateral complete cleft lip and palate presenting 2 impacted supernumerary teeth along the cleft line.

Surgical extraction of both supernumerary teeth was planned. During the procedure, 1 tooth was inadvertently displaced into the nasal cavity. Serial imaging was used to monitor its location and progression.

Assessment of the displaced tooth's trajectory and clinical outcomes through radiographic follow-up.

The displaced tooth migrated from the nasal cavity to the gastrointestinal tract, as confirmed by sequential radiographs. The patient remained asymptomatic throughout and experienced spontaneous elimination of the tooth without complications.

This case highlights the increased risk of tooth displacement during extraction in patients with cleft lip and palate due to compromised anatomical support, and although multidisciplinary perioperative management was essential, the migration of the tooth into the gastrointestinal tract demonstrates that even with careful planning rare adverse events may still occur, reinforcing the value of multidisciplinary case conferences for a consistent treatment plan, as well as preventive strategies such as coordination with otorhinolaryngology, intraoperative nasal endoscopy in high-risk cases, securing the tooth during manipulation and applying gentle traction to minimize the likelihood of such events.

To describe the timing of surgeries, characterize the number of surgical and anesthetic events, and describe the total number and timing of cleft-related and ancillary surgical procedures from birth to late adolescence, stratified by cleft subtype and syndrome status.

Retrospective, cross-sectional clinical review of medical and surgical records.

A tertiary children's hospital with a dedicated Cleft and Craniofacial Surgical Unit in Adelaide, South Australia.

Children aged 0 to 18 years who underwent surgical intervention for orofacial clefts at the Women's and Children's Hospital between 1985 and 2021.

Surgical management of orofacial clefts and ancillary procedures performed under general anesthesia (GA).

Timing and number of cleft-related surgeries; total number of procedures performed under GA; comparisons by syndrome status and cleft subtype.

A total of 746 children were included. The mean age at primary lip repair was 4.8 months, and palate repair was 18.4 months. Alveolar bone grafting was typically performed at a mean age of 129.4 months. Later procedures included lip/nasal revision and orthognathic surgery. The mean number of GA procedures per child was 3.9 (SD ± 3.3), significantly higher in syndromic children (mean 6.5, SD ± 5.3) than nonsyndromic children (mean 3.4, SD ± 2.3). Noncleft-related procedures, such as grommet insertion (45.6%) and dental treatment under GA (28.1%) added to the total surgical burden.

Children with orofacial clefts experience multiple surgical procedures throughout development, especially those with syndromic diagnoses. These findings reinforce the importance of long-term, multidisciplinary planning and provide data to guide cleft care protocols and family support.

To compare coping strategies and maternal emotional styles among mothers of children with cleft lip and palate (CL/P) and mothers of children without CL/P.

Cross-sectional study.

Ahvaz Multidisciplinary Cleft Lip and Palate Team, Speech Therapy Clinics in Ahvaz.

Mothers of children with CL/P (n = 66); mothers of children without CL/P (n = 59); total n = 125.

The Coping Inventory for Stressful Situations (CISS) and Maternal Emotional Styles Questionnaire (MESQ) were completed by mothers of children with CL/P and children without CL/P.

Coping strategies and emotional styles of mothers of children with CL/P.

The results showed that after controlling for the effect of income, there was a significant difference between the coping styles and emotional styles of the 2 groups of mothers. Mothers of children with CL/P had an emotion-oriented style when faced with stressful situations, and their way of dealing with their children's emotions was an emotional dismissive style. While mothers of children without CL/P had task-oriented and avoidance-oriented style when faced with stressful situations, and their way of dealing with their children's emotions was an emotional coaching style.

The results show that mothers of children with CL/P use less effective coping styles in stressful situations and also often deal with their children's emotions in a dismissive manner. Therefore, due to the important role of mother-child interaction in the process of multidisciplinary assessment and treatment of children with CL/P, it is better to consider the psychological and emotional state of their mothers.

Oronasal fistula (ONF) rates after primary palatoplasty may be influenced by technique improvements and care expansion. We evaluated the temporal and geospatial patterns of ONF.

A systematic review and meta-analysis were performed of publications between January 1960 and November 2024 that reported fistula rate following cleft palate repair.

N/A.

Patients with cleft palate.

Primary palatoplasty.

Fistula rate.

223 publications with 71,238 patients were included. Median fistula rates decreased from the 1960s (13.9%, n = 2) to the 2000s (4.9%, IQR: 2.9%-11.0%). Fistula rates increased significantly in the 2010s (6.7%, IQR: 2.6–14.0%), and continued to increase in the 2020s (10.7%, IQR: 5.7–20.5%, p = 0.005). Fistula rates among European nations showed the largest increase from the 2000s (4.4%, IQR: 2.0–12.5%) to the 2020s (21.1%, IQR: 12.7–49.4%, p = 0.041).

Reported fistula incidence declined from reports in the 1960s through the 2000s but has since increased.

DDX3X-related neurodevelopmental disorder (DDX3X-NDD) is a rare genetic condition that primarily affects females, leading to developmental delays and intellectual disability. DDX3X variants, primarily de novo, account for an estimated 1% to 3% of females with unknown causes of intellectual disability. Affected males have also been reported, often with the variant being inherited from an asymptomatic or mildly symptomatic mother.

Case 1. The patient is a 22-month-old female, born full-term, with a history of developmental delay and complete bilateral cleft lip and palate (CLP). Whole exome sequencing (WES) identified a de novo pathogenic missense variant in the DDX3X gene, DDX3X c.1039G > T (p.Asp347Tyr). Developmentally, the patient is progressing gradually—can sit unassisted, but remains nonverbal.

Case 2. The patient is a 4-year-old female, born full-term, with a history of right hemifacial microsomia and DDX3X-NDD, and no known family history. The patient has global developmental delay, but is able to speak in 3- to 4-word sentences. WES identified a de novo DDX3X frameshift variant, c.841_842delC>A (p.Gln281AspfsX13). Facial features include right-sided hemifacial microsomia, with right enophthalmos, microtia, mandibular hypoplasia, and bilateral facial nerve weakness. The patient is well-managed at home and receives regular care from neurology and speech therapy at school.

Although there have been numerous reports on the neurodevelopmental aspects of DDX3X-NDD, craniofacial findings such as hemifacial microsomia and CLP have been minimally reported in the literature. Given the association between DDX3X variants and craniofacial findings, plastic surgeons and their multidisciplinary team should be aware of this genetic condition.

To present a comprehensive guide for integrating digital scanning into the presurgical infant orthopedics (PSIO) workflow, enhancing precision and efficiency in cleft lip and palate (CLP) treatment.

Descriptive study

The guide emphasizes the use of high-precision intraoral scanners and essential armamentarium for effective scanning. Key aspects include infant and operator positioning, secure infant stabilization, and ergonomic considerations for clinicians. Different scanning trajectories are recommended to ensure complete anatomical capture.

High-resolution digital models generated through meticulous preparation and iterative scanning techniques mitigate potential pitfalls, such as motion artifacts and incomplete scans. These models enable the development of a digital workflow for creating patient-specific appliances.

Integrating digital scanning into the PSIO workflow improves the accuracy, efficiency, and effectiveness of CLP treatment in clinical settings.

To identify, describe, and characterize computer-aided design and computer-aided manufacturing (CAD/CAM) methods for nasoalveolar molding (NAM) based on a structured search of scientific literature.

Scoping review was conducted following the PRISMA-ScR guidelines. Searches were done in MEDLINE, Embase, Web of Science, Cochrane Library, and Scopus. Screening and data extraction were performed.

Infants with unrepaired, nonsyndromic, complete unilateral cleft lip and palate (UCLP), or bilateral cleft lip and palate (BCLP).

CAD/CAM NAM.

Outcome measures were the digitization, virtual modeling, and manufacturing protocols.

Thirteen articles were included. CAD/CAM NAM involved digitizing the maxilla, designing step-by-step (stepwise) alveolar movements or expansion of plates, then manufacturing plates manually or through 3D printing. Four methods were characterized based on the virtual modeling and manufacturing techniques employed: stepwise alveolar molding and manually fabricated plates (SM_MP); stepwise alveolar molding and 3D-printed plates (SM_3P); stepwise plate expansion and 3D-printed plates (SPE_3P); and semi-automated plate expansion and 3D-printed plates (SAPE_3P). The SM_MP method was the most common, followed by the SM_3P, SPE_3P, and SAPE_3P methods. All methods were applied to treat infants with UCLP, whereas only the SM_MP and SM_3P methods were used for infants with BCLP.

This scoping review provides an overview of 4 CAD/CAM methods for NAM. The SM_MP and SM_3P methods simulate alveolar molding; however, the SM_3P method exhibits more advanced design and manufacturing of plates. The SPE_3P and SAPE_3P methods design consecutively enlarged plates, with the latter employing a semi-automated protocol.